Introduction: Anterior segment duplicity with two lenses is a rare event which pathogenesis is still unknown. Different ocular and systemic abnormalities might be associated with such event. Case observations: Hereby we describe a case of a 6-months female child referred to our service due to signs of ocular malformation in the left eye. The ocular exam showed a double anterior segment with twin lenses in one single eye, associated with double lamellar opacity and persistence of the two hyaloid arteries. The patient underwent surgical treatment with lensectomy and vitrectomy aiming visual stimulation and prevention of definitive visual loss secondary to amblyopia. Conclusion: In any case of ocular malformation, efforts to provide adequate visual stimulus are necessary to avoid amblyopia. In our case, the opacified lenses were removed, the patient adapted contact lenses and will be followed-up for visual stimulation at the ophthalmic pediatric division aiming the best visual prognosis possible.
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